Multimodal treatment of radiation-associated laryngeal angiosarcoma: A case report and literature review.
Yiyun Pan, Xiaomei Liu, Xiaoming Nie, Shengkun Wang, Wen Zeng
Abstract
Open AccessRATIONALE: Angiosarcoma is a rare, highly aggressive malignancy of vascular endothelial origin, marked by rapid local invasion and a high risk of distant spread. Radiation-associated disease is even rarer, and laryngeal involvement is scarcely reported. No standard therapy exists for locally advanced or metastatic cases, and clinical experience is limited. We report a case of radiation-associated laryngeal angiosarcoma and contextualize its diagnosis and management within the literature. PATIENT CONCERNS: The patient, a 31-year-old man, was admitted to the hospital, at January 24, 2022, due complaints of generalized bone pain, predominantly in the ribs and lumbar spine, worsening with coughing, along with hoarseness and episodes of choking while drinking, persisting for over 1 month. DIAGNOSES: Malignant tumor of the larynx (T3N0M1, stage IVc, AJCC 7th edition); bone metastatic malignancy; splenic metastatic disease; and history of nasopharyngeal carcinoma. INTERVENTIONS: Initial treatment with liposomal doxorubicin plus ifosfamide provided partial symptom relief. Genomic testing identified a TP53 mutation, and toripalimab with gemcitabine/nab-paclitaxel produced a partial response lasting 9 months. After progression, sequential targeted agents (anlotinib, lenvatinib, bevacizumab, pazopanib) did not maintain disease control, and the patient died of progressive disease. OUTCOMES: The patient died on October 5, 2024, due to respiratory failure, bone marrow suppression, and uncontrolled systemic pain. LESSONS: Radiation-associated laryngeal angiosarcoma is exceptionally rare and portends a poor prognosis. This case illustrates the potential value of genomically informed multimodal therapy integrating chemotherapy, immunotherapy, and targeted agents; in our patient, toripalimab plus gemcitabine/nab-paclitaxel achieved a 9-month partial response. Further studies are needed to establish evidence-based treatment strategies for this understudied disease.